Mild hemolytic anemia, progressive neuromotor retardation and fatal outcome: a disorder of glycolysis, triose- phosphate isomerase deficiency.
نویسندگان
چکیده
A two-month-old male infant presented with jaundice, pallor, and hepatomegaly. The first child of non-consanguineous parents had also suffered from hemolytic anemia and neuromotor retardation and died at the age of 21 months. The patient required phototherapy and transfusion in the newborn period but hemolysis was mild thereafter. The patient had neuromotor retardation, and at the age of 14 months, ventilatory support was necessary, and the patient lived until 17 months. Triose-phosphate isomerase (TPI) deficiency, which is a rare autosomal recessive multisystem disorder of glycolysis, was detected. There was homozygous missense mutation in the TPI1 gene (p.Glu105Asp). This is the most common mutation with a severe phenotype that requires ventilator support in the second year of life. In patients with hemolysis and neuromotor retardation, TPI deficiency must be considered. There is no specific treatment, but detection of the index case may provide the opportunity for genetic counseling and prenatal diagnosis.
منابع مشابه
Reversal of metabolic block in glycolysis by enzyme replacement in triosephosphate isomerase-deficient cells.
Inherited deficiency of the housekeeping enzyme triosephosphate isomerase (TPI) is the most severe clinical disorder of glycolysis. Homozygotes manifest congenital hemolytic anemia and progressive neuromuscular impairment, which in most cases pursues an inexorable course with fatal outcome in early childhood. No effective therapy is available. Hitherto specific enzyme replacement has not been a...
متن کاملDegradation of functional triose phosphate isomerase protein underlies sugarkill pathology.
Triose phosphate isomerase (TPI) deficiency glycolytic enzymopathy is a progressive neurodegenerative condition that remains poorly understood. The disease is caused exclusively by specific missense mutations affecting the TPI protein and clinically features hemolytic anemia, adult-onset neurological impairment, degeneration, and reduced longevity. TPI has a well-characterized role in glycolysi...
متن کاملTitle: Degradation of Functional Tpi Protein Underlies Sugarkill Pathology
TPI deficiency glycolytic enzymopathy is a progressive neurodegenerative condition that remains poorly understood. The disease is caused exclusively by specific missense mutations affecting the triose phosphate isomerase protein (TPI) and clinically features hemolytic anemia, adult-onset neurological impairment, degeneration, and reduced longevity. TPI has a well-characterized role in glycolysi...
متن کاملDetection of glucose-phosphate isomerase deficiency by a screening procedure.
G LUCOSE-PHOSPHATE ISOMERASE (GPI) controls the equilibrium between glucose-6-phosphate (G-6-P) and fructose-6-phosphate (F-6-P). Though G?I does not seem to have a rate-limiting function in red cell glycolysis,’ deficiencies of this enzyme cause hereditary nonspherocytic hemolytic anemia.2 Twelve cases from nine different families have been reported suffering from hemolytic anemia caused by G?...
متن کاملHuman triose-phosphate isomerase deficiency: a single amino acid substitution results in a thermolabile enzyme.
Triose-phosphate isomerase (TPI; D-glyceraldehyde-3-phosphate ketol-isomerase, EC 5.3.1.1) deficiency is a recessive disorder that results in hemolytic anemia and neuromuscular dysfunction. To determine the molecular basis of this disorder, a TPI allele from two unrelated patients homozygous for TPI deficiency was compared with an allele from a normal individual. Each disease-associated sequenc...
متن کاملذخیره در منابع من
با ذخیره ی این منبع در منابع من، دسترسی به آن را برای استفاده های بعدی آسان تر کنید
برای دانلود متن کامل این مقاله و بیش از 32 میلیون مقاله دیگر ابتدا ثبت نام کنید
ثبت ناماگر عضو سایت هستید لطفا وارد حساب کاربری خود شوید
ورودعنوان ژورنال:
- The Turkish journal of pediatrics
دوره 55 2 شماره
صفحات -
تاریخ انتشار 2013